TPM2

Chr 9

tropomyosin 2

Also known as: AMCD1, CMYO23, CMYP23, DA1, DA2B, DA2B4, HEL-S-273, NEM4

Beta-tropomyosin is an actin filament binding protein primarily expressed in slow, type 1 muscle fibers that regulates muscle contraction. Mutations cause autosomal dominant congenital myopathies including cap disease and nemaline myopathy, as well as distal arthrogryposis syndromes types 1A and 2B4. The pathogenic mechanism involves altered expression of other sarcomeric tropomyosin proteins through a dominant-negative effect.

ResearchSummary from RefSeq, OMIM, UniProt, Mechanism
MultiplemechanismLOEUF 0.83
Clinical SummaryTPM2
🧬
Gene-Disease Validity (ClinGen)
TPM2-related myopathy · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
Some data sources returned errors (1)

omim: Error: OMIM fetch failed: 429

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.83LOEUF
pLI 0.000
Z-score 2.12
OE 0.47 (0.280.83)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
2.13Z-score
OE missense 0.52 (0.430.62)
80 obs / 154.7 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.47 (0.280.83)
00.351.4
Missense OE0.52 (0.430.62)
00.61.4
Synonymous OE1.03
01.21.6
LoF obs/exp: 9 / 19.0Missense obs/exp: 80 / 154.7Syn Z: -0.19
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongTPM2-related arthrogryposis, distalOTHERAD
DN
0.93top 5%
GOF
0.6832th %ile
LOF
0.1697th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median · 1 literature citation
GOFprediction above median · 1 literature citation

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

Literature Evidence

DNStudies of patient muscle and differentiated myotubes transfected with the mutation suggested that the mutant protein incorporates poorly into sarcomeres and likely accumulates in nemaline bodies, and interferes with wildtype in a dominant-negative manner.PMID:23378224
GOFMutations in repeating structural motifs of tropomyosin cause gain of function in skeletal muscle myopathy patientsPMID:23886664

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

TPM2 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →